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Summary of the impact

Around 1,000 children are born each year in the UK with cleft lip and/or
palate. They need treatment from a range of clinical specialties. Bristol
co-led a research survey commissioned by the Clinical Standards Advisory
Group (CSAG) which was published in 1998. This informed a process of
centralisation which by 2008 had reduced the number of centres from 57 to
11. The process of centralisation and service configuration has continued
through 2008-13 leading to closer multi-disciplinary working, increased
cases operated on per surgeon, less variation in surgical techniques and
shorter stays in hospital. The impact for the individual is improved
facial appearance, speech and psychological adjustment. This
centralisation of care has resulted in care quality similar to the better
cleft centres in Europe. Bone repairs to the palate were 85% successful in
2010 compared to only 58% in 1998 and there was poor jaw growth (linked to
quality of surgery) in only 18% of cases in 2013 compared to 37% in 1998.
The reach of this study has extended to several European countries and
America.

Underpinning research

Cleft lip and palate is a common congenital anomaly in humans, with
issues that relate to appearance, speech, hearing, general health and
social integration requiring a range of healthcare professional skills. In
the late 1980's and early 1990's there were indications that care for
children born with a cleft lip and palate in the UK was varied and often
suboptimal. Based on this, the Royal College of Surgeons Audit Unit and
the Standing Dental Advisory Committee made appeals to the Department of
Health that a Clinical Standards Advisory Group (CSAG) enquiry be
established to determine national levels of care for those born with a
cleft. The CSAG committee commissioned a research team to carry out a
survey.

Logistics of the research survey
The research team was co-led by Jonathan Sandy (Professor in Orthodontics
at U. Bristol, 1991 onwards) and Bill Shaw (Professor in Orthodontics at
U. Manchester), both with track records in cleft research. The research
described the care and outcome in non-syndromic cases of unilateral cleft
lip and palate (UCLP) aged five and twelve years throughout the UK treated
over a two year period. Information was collected on the process of care
and key outcomes were measured including jaw growth, bone grafting, facial
appearance, speech, hearing, dental health and child/parent satisfaction.
Thus high quality research was used to inform decisions on the service.
At that time there were 57 cleft centres operating on these children
which required the researchers to drive a total of 40,000 miles to collect
all of the data. Two part time research registrars (Alison Williams [RA,
PhD student and Clinician Scientist Award at U. Bristol, 1996-2003] and
David Bearn [Manchester] and two part time speech and language therapists
(Sue Mildinhall [RA at Bristol 1995 -1997] and Terrie Murphy [Manchester]
were appointed to collect and analyse the data. Bristol wrote the grant,
developed the protocol, collected half of the data and led on the analysis
and publication of the data.

Outcomes and cleft care in the UK
The research survey published in 1998 showed for the first time that the
outcomes nationally were sub-optimal [1]. For example, poor growth of the
top jaw was present in 37% of five year olds and 39% of twelve year olds.
The implication of this finding was that around 40% of children were
likely to need an operation to break, and move, the top jaw forward to
correct an underlying discrepancy between the top and bottom jaws [2].
This compared with around 4% in children treated in leading European
centres where this growth was better. Another example of poor outcome was
seen in bone grafting in the upper jaw. This is a procedure undertaken
around the age of 11 to unite the divided upper jaw and allow eruption of
adjacent permanent teeth. There are very few, if any, children with a UCLP
who would not be offered this operation. However, 16% of the UK twelve
year olds had not received this bone graft, and of those who had a graft,
only 58% were successful compared to a 92% success rate in some of the
better European centres [2].

The predominance of low volume operators (nearly 60% of surgeons were
dealing with only one UCLP case per year) and overall poor quality of
results limited the detailed exploration of associations between volume
and outcome but the survey suggested the more operations a surgeon did the
better the results. There was thus evidence that the service needed to be
centralised. The results were published as a report [1] (can be supplied
upon request) and a series of papers [2, 4-6] in the only specialist
journal for cleft clinicians and one in a leading general medical journal
[3]. A grant was subsequently awarded (2009) by the NIHR to Andrew Ness
(Professor in Epidemiology at U. Bristol, 2007 onwards) to examine the
impact of centralisation of cleft services. Data collection is now
complete and early analysis of poor growth of the top jaw (which was
present in 37% of five year olds in the original CSAG) suggests this has
fallen by 50%.

Details of the impact

The CSAG committee prepared a report (Section 3, ref [1]) based on the
evidence collected by the research team (co-led by Jonathan Sandy). The
recommendations of this report were accepted by Ministers (Health Services
Circular [HSC] 1998/002) and the Department of Health announced the
establishment of the cleft implementation group (CIG) in HSC 1998/087.
This was the first time that a CSAG report had been followed by a formal
process of implementation and included the development and maintenance of
a national database (CRANE) to record the births of children with a cleft
as well as key audit points with established care indicators [a]. There
were several reasons for this besides the strength and scientific
robustness of the evidence collected. These included the fact that there
was unanimous clinical support for service rationalisation and that the
proposed changes were supported by an active and well informed
user/clinician group (Cleft Lip and Palate Association, CLAPA). The speed
of centralisation varied with the earliest of the nine regions
reconfiguring services in 2001 and the last in 2008. The process of
centralisation has continued through 2008 — 13 as centres have recruited
recommended specialist staff, developed team working and set up
multicentre audits. Most of the CSAG recommendations have now been
implemented and care across the UK is now provided through
multidisciplinary teams [b]. For the individual, this means better care
resulting in improvements in facial appearance, speech and ultimately
psychological adjustment. The number of surgeons responsible for repairs
reduced from 98 before centralisation to 26 in 2008. Furthermore 92% of
cleft units operate on at least 40 patients a year compared to an average
of 6 per year before CSAG centralisation [c]. The extensive research also
identified areas of care that lacked provision within the service. These
deficiencies were highlighted in the CSAG report and as centralisation
moved forward, strong arguments have been made to improve staff and
capital resources in the newly designated centres. The nature and extent
of the impact is becoming apparent with preliminary evidence that
outcomes, collected through audits set up as part of the implementation of
the CSAG report, have improved. Success rates for upper jaw bone grafting
were 85% in 2010 compared to 58% pre- CSAG [d]. In addition, less than 20%
of children have poor upper and lower jaw relations when treated within a
centralised service compared to 40% pre-CSAG 40% [e]. Several
geographically proximate regions have come together to run regular
tri-centre audits to review outcomes. This demonstrates a commitment to
review the outcomes and evaluate the service through continued audit.
Early nursing input at diagnosis before or after birth has improved
immeasurably post centralisation as has outreach nursing and feeding
support [f].

User awareness of the impact of CSAG
In 2006 CLAPA surveyed parents of children born with a cleft between 2002
and 2005. This was similar to the survey carried out pre-CSAG in 1996. The
2007 report recognised that the changes in cleft care over the last five
years mean that the needs of families and children born with clefts are
better catered for than ever before. Ten years ago it was still largely
other parents, under the umbrella of CLAPA, who guided and advised on some
of the early care, especially around feeding. Specialist cleft nurses now
provide that early care and tried and tested surgical protocols are in
place meaning that babies receive surgery to a high standard, provided by
a surgeon who has well documented expertise and experience [g]. A further
survey of members by CLAPA in 2010 suggested that there had been further
improvements in care. The survey reported that there was now a closer
relationship with teams enabling patient representatives to be part of the
team. Furthermore, CLAPA had helped draft the new National Service
Specification as well as engaging media on issues such as late diagnosis
of cleft palate [h].

Impact on service commissioning and specification in the UK
The NHS commissioning board in issuing the 2012/13 NHS standard contract
(service specification D7) for Cleft Lip and Palate Services uses the 1998
CSAG report to summarise the key issues and challenges to be addressed by
providers and commissioners [i]. The specification builds on the
recommendations and does not replicate or supersede the original CSAG
report. This formally recognises the importance of the CSAG research and
its central role in policy formation. The USA national medical policy
(2011) uses CSAG data to inform service configuration [j]. These latter
policies are based upon a review of the available clinical information
including clinical outcome studies in the peer-reviewed published medical
literature and evidence-based guidelines of governmental bodies.

Global impact
The CSAG study was widely recognised as an important step in the
evaluation of cleft care services including the WHO in their documentation
of global strategies to reduce the health-care burden of craniofacial
anomalies (2001). After CSAG, other countries became interested in
conducting similar surveys. The Americleft study has produced results from
one centre that are worse than any of the pre-CSAG UK centres. There is
now an inevitable process of centralisation with care being moved towards
University Hospitals [k]. The data from CSAG provided a basis and logic
for centralisation which a number of countries used without gathering the
same level of information. There is evidence in Europe that the influence
of the original CSAG has led to the creation of centres of excellence in
France (2013) which has been driven by the Ministry of Health (http://www.fente-labio-palatine.fr/).
In Bulgaria and Romania similar service recommendations have been made
with single high volume units, specialist speech therapists and national
registers (2012). These decisions used the CSAG data to inform their
decisions.